Optimal surgical treatment for paratesticular leiomyosarcoma: Retrospective analysis of 217 reported cases

Paratesticular leiomyosarcoma (LMS) is a rare tumor. Conventionally, tumor resection by high inguinal orchiectomy is performed as the preferred treatment approach for paratesticular sarcoma. On the other hand, testis-sparing surgery has recently attracted attention as a less-invasive treatment option for paratesticular sarcoma.
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Optimal surgical treatment for paratesticular leiomyosarcoma: Retrospective analysis of 217 reported casesKamitanietal. BMC Cancer (2022) 22:15https://doi.org/10.1186/s12885-021-09122-7 RESEARCH Open AccessOptimal surgical treatment forparatesticularleiomyosarcoma: retrospective analysis of217reported casesReiKamitani, KazuhiroMatsumoto*, ToshikazuTakeda, RyuichiMizunoand MototsuguOya  Abstract  Background:  Paratesticular leiomyosarcoma (LMS) is a rare tumor. Conventionally, tumor resection by high ingui- nal orchiectomy is performed as the preferred treatment approach for paratesticular sarcoma. On the other hand, testis-sparing surgery has recently attracted attention as a less-invasive treatment option for paratesticular sarcoma. However, the prognostic predictors and optimal treatment strategy for paratesticular LMS remain unclear because of its rarity. In this study, we systematically reviewed previously reported cases of paratesticular LMS to evaluate the prognostic factors and establish the optimal treatment strategy. Methods:  A systematic search of Medline, Web of Science, Embase, and Google was performed to find articles describing localized paratesticular LMS published between 1971 and 2020 in English. The final cohort included 217 patients in 167 articles. The starting point of this study was the time of definitive surgical treatment, and the end point was the time of local recurrence (LR), distant metastasis (DM), and disease-specific mortality. Results:  Patients with cutaneous LMS had a slightly better LR-free survival, DM-free survival, and disease-specific sur- vival than those with subcutaneous LMS (p = 0.745, p = 0.033, and p = 0.126, respectively). Patients with higher grade tumors had a significantly higher risk of DM and disease-specific mortality (Grade 3 vs Grade 1 p Kamitanietal. BMC Cancer (2022) 22:15 Page 2 of 9Among genitourinary STSs, leiomyosarcoma (LMS) is described in different reports were treated as a singleone of the common histological subtypes that sometimes case. All searches were conducted independently by twodevelops in the paratesticular region [2–4]. Possible ori- authors (RK and KM). The results were compared, andgins of paratesticular LMS are intratesticular seminifer- questions or discrepancies were resolved through itera-ous tubules, the epididymis, spermatic cord, dartos layer, tion and consensus. The study flow diagram is shown inand scrotal skin. According to the location of origin, it is Fig.1. In total, 167 articles fulfilled our inclusion criteriadivided into 2 types: cutaneous LMS that originates from and provided a total of 217 cases for systematic reviewthe arrector pili muscle of hair follicle or dartos muscle of (Supplemental Table1).genital skin, and subcutaneous LMS that originates from The collected clinical data included age, laterality,smooth muscle of genital organ or the vascular muscle tumor size, tumor depth, surgical treatment, tumorlayer of subcutaneous tissue [5, 6]. grade, surgical margin, adjuvant treatment, and clinical The clinical practice guidelines for STS recommend outcomes. Regarding tumor depth, the cases were dividedcomplete surgical resection including the surround- into 2 groups; cutaneous LMS and subcutaneous LMS [5,ing tissue to achieve an appropriate margin status as 6]. We regarded LMS that infiltrated subcutaneous tissuethe standard treatment [7–10]. However, the specific as subcutaneous LMS. Regarding tumor grade, the casestreatment strategy for paratesticular LMS has not been were divided into 3 groups, Grade 1 (low-grade), Grade 2established. Conventionally, tumor resection with high (intermediate-grade), and Grade 3 (high-grade), accord-inguinal orchiectomy is performed as the preferred treat- ing to the National Federation of French Cancer Centerment approach for paratesticular STS [11–13]. On the Institute System, a scoring system based on the evalua-other hand, testis-sparing surgery has recently attracted tion of the number of mitoses, percentage of necrosis,attention as a less-invasive treatment option for parates- and severity of nuclear pleomorphism [20, 21].ticular STS [14–16]. Previous case reports of paratesticu- The starting point of this study was the time of defini-lar LMS demonstrated good disease control even when tive surgical treatment, and the end point was the timetreated by simple tumorectomy sparing the testis [5, of local recurrence (LR), distant metastasis (DM), and6, 17, 18]. However, the prognostic factors and optimal disease-specific mortality. Categorical variables weretreatment strategy for paratesticular LMS remain unclear compared using the two-sided Fischer’s test and continu-because of its rarity. ous variables were compared using the Mann-Whitney Regarding liposarcoma, which is another of com- U-test. The LR-free, DM-free, and disease-specific sur-mon histological subtype of genitourinary STS, we pre- vival (DSS) curves were constructed using the Kaplan-viously performed a systematic review of case reports Meier method and compared by the log-rank test. Toand revealed that complete resection with high inguinal determine risk factors for LR and DM, multivariate anal-orchiectomy is beneficial [19]. Similar to p ...